Search Results

You are looking at 1 - 10 of 144 items for

  • Abstract: Calcium x
  • Abstract: Hyperparathyroidism x
  • Abstract: Hypoparathyroidism x
  • Abstract: Menopause x
  • Abstract: Osteo* x
  • Abstract: Skeleton x
Clear All Modify Search
Athanasios D Anastasilakis Department of Endocrinology, 424 General Military Hospital, Thessaloniki, Greece

Search for other papers by Athanasios D Anastasilakis in
Google Scholar
PubMed
Close
,
Marina Tsoli 1st Propaedeutic Department of Internal Medicine, National and Kapodistrian University of Athens, Athens, Greece

Search for other papers by Marina Tsoli in
Google Scholar
PubMed
Close
,
Gregory Kaltsas 1st Propaedeutic Department of Internal Medicine, National and Kapodistrian University of Athens, Athens, Greece

Search for other papers by Gregory Kaltsas in
Google Scholar
PubMed
Close
, and
Polyzois Makras Department of Endocrinology and Diabetes, 251 Hellenic Air Force & VA General Hospital, Athens, Greece

Search for other papers by Polyzois Makras in
Google Scholar
PubMed
Close

Langerhans cell histiocytosis (LCH) is a rare disease of not well-defined etiology that involves immune cell activation and frequently affects the skeleton. Bone involvement in LCH usually presents in the form of osteolytic lesions along with low bone mineral density. Various molecules involved in bone metabolism are implicated in the pathogenesis of LCH or may be affected during the course of the disease, including interleukins (ILs), tumor necrosis factor α, receptor activator of NF-κB (RANK) and its soluble ligand RANKL, osteoprotegerin (OPG), periostin and sclerostin. Among them IL-17A, periostin and RANKL have been proposed as potential serum biomarkers for LCH, particularly as the interaction between RANK, RANKL and OPG not only regulates bone homeostasis through its effects on the osteoclasts but also affects the activation and survival of immune cells. Significant changes in circulating and lesional RANKL levels have been observed in LCH patients irrespective of bone involvement. Standard LCH management includes local or systematic administration of corticosteroids and chemotherapy. Given the implication of RANK, RANKL and OPG in the pathogenesis of the disease and the osteolytic nature of bone lesions, agents aiming at inhibiting the RANKL pathway and/or osteoclastic activation, such as bisphosphonates and denosumab, may have a role in the therapeutic approach of LCH although further clinical investigation is warranted.

Open access
Elinor Chelsom Vogt Department of Clinical Science, University of Bergen, Bergen, Norway
K.G. Jebsen Center for Autoimmune Disorders, University of Bergen, Bergen, Norway
Department of Medicine, Haukeland University Hospital, Bergen, Norway

Search for other papers by Elinor Chelsom Vogt in
Google Scholar
PubMed
Close
,
Francisco Gómez Real Department of Clinical Science, University of Bergen, Bergen, Norway
Department of Gynecology and Obstetrics, Haukeland University Hospital, Bergen, Norway

Search for other papers by Francisco Gómez Real in
Google Scholar
PubMed
Close
,
Eystein Sverre Husebye Department of Clinical Science, University of Bergen, Bergen, Norway
K.G. Jebsen Center for Autoimmune Disorders, University of Bergen, Bergen, Norway
Department of Medicine, Haukeland University Hospital, Bergen, Norway

Search for other papers by Eystein Sverre Husebye in
Google Scholar
PubMed
Close
,
Sigridur Björnsdottir Department of Molecular Medicine and Surgery, Karolinska Institutet, Stockholm, Sweden
Department of Endocrinology, Metabolism and Diabetes, Karolinska University Hospital, Stockholm, Sweden

Search for other papers by Sigridur Björnsdottir in
Google Scholar
PubMed
Close
,
Bryndis Benediktsdottir Medical Faculty, University of Iceland, Reykjavik, Iceland
Department of Sleep, Landspitali University Hospital Reykjavík, Reykjavik, Iceland

Search for other papers by Bryndis Benediktsdottir in
Google Scholar
PubMed
Close
,
Randi Jacobsen Bertelsen Department of Clinical Science, University of Bergen, Bergen, Norway

Search for other papers by Randi Jacobsen Bertelsen in
Google Scholar
PubMed
Close
,
Pascal Demoly University Hospital of Montpellier, IDESP, Univ Montpellier-Inserm, Montpellier, France

Search for other papers by Pascal Demoly in
Google Scholar
PubMed
Close
,
Karl Anders Franklin Department of Surgical and Perioperative Sciences, Surgery, Umeå University, Umeå, Sweden

Search for other papers by Karl Anders Franklin in
Google Scholar
PubMed
Close
,
Leire Sainz de Aja Gallastegui Unit of Epidemiology and Public Health, Department of Health, Basque Government, Vitoria-Gasteiz, Spain

Search for other papers by Leire Sainz de Aja Gallastegui in
Google Scholar
PubMed
Close
,
Francisco Javier Callejas González Department of Respiratory Medicine, Albacete University Hospital, Albacete, Spain

Search for other papers by Francisco Javier Callejas González in
Google Scholar
PubMed
Close
,
Joachim Heinrich Institute and Clinic for Occupational, Social and Environmental Medicine, University Hospital, LMU Munich, Munich, Germany
Allergy and Lung Health Unit, Melbourne School of Population and Global Health, The University of Melbourne, Melbourne, Victoria, Australia

Search for other papers by Joachim Heinrich in
Google Scholar
PubMed
Close
,
Mathias Holm Occupational and Environmental Medicine, School of Public Health and Community Medicine, Institute of Medicine, Sahlgrenska Academy, University of Gothenburg, Gothenburg, Sweden

Search for other papers by Mathias Holm in
Google Scholar
PubMed
Close
,
Nils Oscar Jogi Department of Clinical Science, University of Bergen, Bergen, Norway

Search for other papers by Nils Oscar Jogi in
Google Scholar
PubMed
Close
,
Benedicte Leynaert Université Paris-Saclay, Inserm U1018, Center for Epidemiology and Population Health, Integrative Respiratory Epidemiology Team, Villejuif, France

Search for other papers by Benedicte Leynaert in
Google Scholar
PubMed
Close
,
Eva Lindberg Department of Medical Sciences, Respiratory, Allergy and Sleep Medicine, Uppsala University, Uppsala, Sweden

Search for other papers by Eva Lindberg in
Google Scholar
PubMed
Close
,
Andrei Malinovschi Department of Medical Sciences, Clinical Physiology, Uppsala University, Uppsala, Sweden

Search for other papers by Andrei Malinovschi in
Google Scholar
PubMed
Close
,
Jesús Martínez-Moratalla Pneumology Service of the General University Hospital of Albacete, Albacete, Spain
Albacete Faculty of Medicine, Castilla-La Mancha University, Albacete, Spain

Search for other papers by Jesús Martínez-Moratalla in
Google Scholar
PubMed
Close
,
Raúl Godoy Mayoral Department of Respiratory Medicine, Albacete University Hospital, Albacete, Spain

Search for other papers by Raúl Godoy Mayoral in
Google Scholar
PubMed
Close
,
Anna Oudin Section of Sustainable Health, Department of Public Health and Clinical Medicine, Umeå University, Umeå, Sweden

Search for other papers by Anna Oudin in
Google Scholar
PubMed
Close
,
Antonio Pereira-Vega Juan Ramón Jiménez University Hospital in Huelva, Huelva, Spain

Search for other papers by Antonio Pereira-Vega in
Google Scholar
PubMed
Close
,
Chantal Raherison Semjen INSERM, EpiCene Team U1219, University of Bordeaux, Talence, France

Search for other papers by Chantal Raherison Semjen in
Google Scholar
PubMed
Close
,
Vivi Schlünssen Department of Public Health, Environment, Work and Health, Danish Ramazzini Centre, Aarhus University, Aarhus, Denmark
The National Research Center for the Working Environment, Copenhagen, Denmark

Search for other papers by Vivi Schlünssen in
Google Scholar
PubMed
Close
,
Kai Triebner Department of Clinical Science, University of Bergen, Bergen, Norway

Search for other papers by Kai Triebner in
Google Scholar
PubMed
Close
, and
Marianne Øksnes Department of Clinical Science, University of Bergen, Bergen, Norway
K.G. Jebsen Center for Autoimmune Disorders, University of Bergen, Bergen, Norway
Department of Medicine, Haukeland University Hospital, Bergen, Norway

Search for other papers by Marianne Øksnes in
Google Scholar
PubMed
Close

Objective

To investigate markers of premature menopause (<40 years) and specifically the prevalence of autoimmune primary ovarian insufficiency (POI) in European women.

Design

Postmenopausal women were categorized according to age at menopause and self-reported reason for menopause in a cross-sectional analysis of 6870 women.

Methods

Variables associated with the timing of menopause and hormone measurements of 17β-estradiol and follicle-stimulating hormone were explored using multivariable logistic regression analysis. Specific immunoprecipitating assays of steroidogenic autoantibodies against 21-hydroxylase (21-OH), side-chain cleavage enzyme (anti-SCC) and 17alpha-hydroxylase (17 OH), as well as NACHT leucine-rich-repeat protein 5 were used to identify women with likely autoimmune POI.

Results

Premature menopause was identified in 2.8% of women, and these women had higher frequencies of nulliparity (37.4% vs 19.7%), obesity (28.7% vs 21.4%), osteoporosis (17.1% vs 11.6%), hormone replacement therapy (59.1% vs 36.9%) and never smokers (60.1% vs 50.9%) (P < 0.05), compared to women with menopause ≥40 years. Iatrogenic causes were found in 91 (47%) and non-ovarian causes in 27 (14%) women, while 77 (39%) women were classified as POI of unknown cause, resulting in a 1.1% prevalence of idiopathic POI. After adjustments nulliparity was the only variable significantly associated with POI (odds ratio 2.46; 95% CI 1.63–3.42). Based on the presence of autoantibodies against 21 OH and SCC, 4.5% of POI cases were of likely autoimmune origin.

Conclusion

Idiopathic POI affects 1.1% of all women and almost half of the women with premature menopause. Autoimmunity explains 4.5% of these cases judged by positive steroidogenic autoantibodies.

Open access
Sofya Gronskaia Endocrinology Research Centre, Moscow, Russia

Search for other papers by Sofya Gronskaia in
Google Scholar
PubMed
Close
,
Galina Melnichenko Endocrinology Research Centre, Moscow, Russia

Search for other papers by Galina Melnichenko in
Google Scholar
PubMed
Close
,
Liudmila Rozhinskaya Endocrinology Research Centre, Moscow, Russia

Search for other papers by Liudmila Rozhinskaya in
Google Scholar
PubMed
Close
,
Tatiana Grebennikova Endocrinology Research Centre, Moscow, Russia

Search for other papers by Tatiana Grebennikova in
Google Scholar
PubMed
Close
,
Elizaveta Mamedova Endocrinology Research Centre, Moscow, Russia

Search for other papers by Elizaveta Mamedova in
Google Scholar
PubMed
Close
,
Ekaterina Pigarova Endocrinology Research Centre, Moscow, Russia

Search for other papers by Ekaterina Pigarova in
Google Scholar
PubMed
Close
,
Elena Przhialkovskaya Endocrinology Research Centre, Moscow, Russia

Search for other papers by Elena Przhialkovskaya in
Google Scholar
PubMed
Close
,
Larisa Dzeranova Endocrinology Research Centre, Moscow, Russia

Search for other papers by Larisa Dzeranova in
Google Scholar
PubMed
Close
,
Ivan Dedov Endocrinology Research Centre, Moscow, Russia

Search for other papers by Ivan Dedov in
Google Scholar
PubMed
Close
,
Valentin Fadeyev I.M. Sechenov First Moscow State Medical University, Moscow, Russia

Search for other papers by Valentin Fadeyev in
Google Scholar
PubMed
Close
,
Maria Luisa Brandi University of Florence, Surgery and Translational Medicine, Piereccaini, Firenze, Italy

Search for other papers by Maria Luisa Brandi in
Google Scholar
PubMed
Close
, and
Zhanna Belaya Endocrinology Research Centre, Moscow, Russia

Search for other papers by Zhanna Belaya in
Google Scholar
PubMed
Close

Hypoparathyroidism and pseudohypoparathyroidism are rare endocrine disorders, characterized by low serum calcium due to inappropriate parathyroid hormone (PTH) levels or resistance to its action. There is little epidemiological information regarding chronic hypoparathyroidism in Russia. This study aims to build a registry database of Russian patients with chronic hypoparathyroidism who were referred for hospital treatment in order to conduct initial analysis of clinical presentations and hospital management. The Italian registry model was taken to be able to integrate our data in the future. Two hundred patients with hypoparathyroidism (n = 194) and pseudohypoparathyroidism (n = 6) were enrolled over 2 years (2017–2019). The most frequent cause of hypoparathyroidism was neck surgery (82.5%, mostly females), followed by idiopathic hypoparathyroidism (10%), syndromic forms of genetic hypoparathyroidism (4.5%) and forms of defective PTH action (3%). Calcium supplements and alfacalcidol were prescribed in most cases. However, a minority of patients (n = 6) needed to receive teriparatide as the only way to maintain calcium levels and to prevent symptoms of hypocalcemia. Consequently, substitution treatment with parathyroid hormone should be available in certain cases of hypoparathyroidism. This database will be useful to estimate the potential requirement for recombinant PTH in Russia and standards for clinical and therapeutic approaches.

Open access
Huda M Elsharkasi Developmental Endocrinology Research Group, University of Glasgow, Glasgow, UK

Search for other papers by Huda M Elsharkasi in
Google Scholar
PubMed
Close
,
Suet C Chen Developmental Endocrinology Research Group, University of Glasgow, Glasgow, UK

Search for other papers by Suet C Chen in
Google Scholar
PubMed
Close
,
Lewis Steell Developmental Endocrinology Research Group, University of Glasgow, Glasgow, UK

Search for other papers by Lewis Steell in
Google Scholar
PubMed
Close
,
Shuko Joseph Developmental Endocrinology Research Group, University of Glasgow, Glasgow, UK
Paediatric Neurosciences Research Group, Royal Hospital for Children, NHS Greater Glasgow & Clyde, Glasgow, UK

Search for other papers by Shuko Joseph in
Google Scholar
PubMed
Close
,
Naiemh Abdalrahaman Developmental Endocrinology Research Group, University of Glasgow, Glasgow, UK

Search for other papers by Naiemh Abdalrahaman in
Google Scholar
PubMed
Close
,
Christie McComb Department of Clinical Physics, NHS Greater Glasgow & Clyde, Glasgow, UK

Search for other papers by Christie McComb in
Google Scholar
PubMed
Close
,
Blair Johnston Department of Clinical Physics, NHS Greater Glasgow & Clyde, Glasgow, UK

Search for other papers by Blair Johnston in
Google Scholar
PubMed
Close
,
John Foster Department of Clinical Physics, NHS Greater Glasgow & Clyde, Glasgow, UK

Search for other papers by John Foster in
Google Scholar
PubMed
Close
,
Sze Choong Wong Developmental Endocrinology Research Group, University of Glasgow, Glasgow, UK

Search for other papers by Sze Choong Wong in
Google Scholar
PubMed
Close
, and
S Faisal Ahmed Developmental Endocrinology Research Group, University of Glasgow, Glasgow, UK

Search for other papers by S Faisal Ahmed in
Google Scholar
PubMed
Close

Objective

The aim of this study is to investigate the role of 3T-MRI in assessing musculoskeletal health in children and young people.

Design

Bone, muscle and bone marrow imaging was performed in 161 healthy participants with a median age of 15.0 years (range, 8.0, 30.0).

Methods

Detailed assessment of bone microarchitecture (constructive interference in the steady state (CISS) sequence, voxel size 0.2 × 0.2 × 0.4 mm3), bone geometry (T1-weighted turbo spin echo (TSE) sequence, voxel size 0.4 × 0.4 × 2 mm3) and bone marrow (1H-MRS, point resolved spectroscopy sequence (PRESS) (single voxel size 20 × 20 × 20 mm3) size and muscle adiposity (Dixon, voxel size 1.1 × 1.1 × 2 mm3).

Results

There was an inverse association of apparent bone volume/total volume (appBV/TV) with age (r = −0.5, P < 0.0005). Cortical area, endosteal and periosteal circumferences and muscle cross-sectional area showed a positive association to age (r > 0.49, P < 0.0001). In those over 17 years of age, these parameters were also higher in males than females (P < 0.05). This sex difference was also evident for appBV/TV and bone marrow adiposity (BMA) in the older participants (P < 0.05). AppBV/TV showed a negative correlation with BMA (r = −0.22, P =  0.01) which also showed an association with muscle adiposity (r = 0.24, P = 0.04). Cortical geometric parameters were highly correlated with muscle area (r > 0.57, P < 0.01).

Conclusions

In addition to providing deep insight into the normal relationships between bone, fat and muscle in young people, these novel data emphasize the role of MRI as a non-invasive method for performing a comprehensive and integrated assessment of musculoskeletal health in the growing skeleton.

Open access
A Chinoy Royal Manchester Children’s Hospital, Manchester, UK

Search for other papers by A Chinoy in
Google Scholar
PubMed
Close
,
M Skae Royal Manchester Children’s Hospital, Manchester, UK

Search for other papers by M Skae in
Google Scholar
PubMed
Close
,
A Babiker King Abdullah Specialized Children’s Hospital, Riyadh, Saudi Arabia

Search for other papers by A Babiker in
Google Scholar
PubMed
Close
,
D Kendall Royal Preston Hospital, Preston, UK

Search for other papers by D Kendall in
Google Scholar
PubMed
Close
,
M Z Mughal Royal Manchester Children’s Hospital, Manchester, UK

Search for other papers by M Z Mughal in
Google Scholar
PubMed
Close
, and
R Padidela Royal Manchester Children’s Hospital, Manchester, UK

Search for other papers by R Padidela in
Google Scholar
PubMed
Close

Background

Hypoparathyroidism is characterised by hypocalcaemia, and standard management is with an active vitamin D analogue and adequate oral calcium intake (dietary and/or supplements). Little is described in the literature about the impact of intercurrent illnesses on calcium homeostasis in children with hypoparathyroidism.

Methods

We describe three children with hypoparathyroidism in whom intercurrent illnesses led to hypocalcaemia and escalation of treatment with alfacalcidol (1-hydroxycholecalciferol) and calcium supplements.

Results

Three infants managed with standard treatment for hypoparathyroidism (two with homozygous mutations in GCMB2 gene and one with Sanjad-Sakati syndrome) developed symptomatic hypocalcaemia (two infants developed seizures) following respiratory or gastrointestinal illnesses. Substantial increases in alfacalcidol doses (up to three times their pre-illness doses) and calcium supplementation were required to achieve acceptable serum calcium concentrations. However, following resolution of illness, these children developed an increase in serum calcium and hypercalciuria, necessitating rapid reduction to pre-illness dosages of alfacalcidol and oral calcium supplementation.

Conclusion

Intercurrent illness may precipitate symptomatic hypocalcaemia in children with hypoparathyroidism, necessitating increase in dosages of alfacalcidol and calcium supplements. Close monitoring is required on resolution of the intercurrent illness, with timely reduction of dosages of active analogues of vitamin D and calcium supplements to prevent hypercalcaemia, hypercalciuria and nephrocalcinosis.

Open access
Guido Zavatta Department of Medical and Surgical Sciences, University of Bologna, Bologna, Italy

Search for other papers by Guido Zavatta in
Google Scholar
PubMed
Close
and
Bart L Clarke Mayo Clinic, Rochester, Minnesota, USA

Search for other papers by Bart L Clarke in
Google Scholar
PubMed
Close

The first adjunctive hormone therapy for chronic hypoparathyroidism, recombinant human parathyroid hormone (1–84) (rhPTH(1–84)) was approved by the FDA in January 2015. Since the approval of rhPTH(1–84), growing interest has developed in other agents to treat this disorder in both the scientific community and among pharmaceutical companies. For several reasons, conventional therapy with calcium and activated vitamin D supplementation, magnesium supplementation as needed, and occasionally thiazide-type diuretic therapy remains the mainstay of treatment, while endocrinologists and patients are constantly challenged by limitations of conventional treatment. Serum calcium fluctuations, increased urinary calcium, hyperphosphatemia, and a constellation of symptoms that limit mental and physical functioning are frequently associated with conventional therapy. Understanding how conventional treatment and hormone therapy work in terms of pharmacokinetics and pharmacodynamics is key to effectively managing chronic hypoparathyroidism. Multiple questions remain regarding the effectiveness of PTH adjunctive therapy in preventing or slowing the onset and progression of the classical complications of hypoparathyroidism, such as chronic kidney disease, calcium-containing kidney stones, cataracts, or basal ganglia calcification. Several studies point toward an improvement in the quality of life during replacement therapy. This review will discuss current clinical and research challenges posed by treatment of chronic hypoparathyroidism.

Key points:

  • Conventional therapy with calcium and activated forms of vitamin D are currently the mainstays of treatment for most patients with chronic hypoparathyroidism.

  • Hormone therapy can be administered through FDA-approved once-daily rhPTH(1–84), or off-label multiple-daily injections of teriparatide. The former is the only FDA-approved drug, with safety and efficacy supported by a randomized placebo-controlled trial and open-label long-term extension trial data.

  • Twice-daily teriparatide has been used in children safely for up to 10 years.

  • New pharmacological options that replace the deficient hormone wi ll likely be available within the next few years.

Open access
Jean-Philippe Bertocchio Assistance Publique-Hôpitaux de Paris, Hôpital Européen Georges Pompidou, Service de Physiologie, Paris, France
Centre de Référence des Maladies Rares du Calcium et du Phosphore Filière de Santé Maladies Rares OSCAR, Paris, France
Centre de Recherche des Cordeliers, INSERM, Sorbonne Université, Université de Paris, INSERM, UMRS1138, Paris, France

Search for other papers by Jean-Philippe Bertocchio in
Google Scholar
PubMed
Close
,
Natalie Grosset Hypoparathyroïdisme France, Annecy, France

Search for other papers by Natalie Grosset in
Google Scholar
PubMed
Close
,
Lionel Groussin Department of Endocrinology, Assistance Publique-Hôpitaux de Paris, Hôpital Cochin, Université de Paris, Paris, France

Search for other papers by Lionel Groussin in
Google Scholar
PubMed
Close
,
Peter Kamenický Université Paris-Saclay, Inserm U1185, Physiologie et Physiopathologie Endocriniennes, Assistance Publique-Hôpitaux de Paris, Hôpital Bicêtre, Service d’Endocrinologie et des Maladies de la Reproduction, Centre de Référence des Maladies Rares du Métabolisme du Calcium et du Phosphate, Le Kremlin-Bicêtre, France

Search for other papers by Peter Kamenický in
Google Scholar
PubMed
Close
,
Fabrice Larceneux Université Paris-Dauphine, PSL Research University, CNRS, UMR 7088, DRM [Ermes], Paris, France

Search for other papers by Fabrice Larceneux in
Google Scholar
PubMed
Close
,
Anne Lienhardt-Roussie CHU Dupuytren, Hôpital Mère Enfant, Endocrinologie Pédiatrique, Limoges, France

Search for other papers by Anne Lienhardt-Roussie in
Google Scholar
PubMed
Close
,
Agnès Linglart Centre de Référence des Maladies Rares du Calcium et du Phosphore Filière de Santé Maladies Rares OSCAR, Paris, France
Université Paris-Saclay, Inserm U1185, Physiologie et Physiopathologie Endocriniennes, Assistance Publique-Hôpitaux de Paris, Service d’Endocrinologie et Diabète de l’Enfant, Centre de Référence des Maladies Rares du Calcium et du Phosphore et Filière de Santé Maladies Rares OSCAR, Hôpital Bicêtre Paris Saclay, Le Kremlin-Bicêtre, France

Search for other papers by Agnès Linglart in
Google Scholar
PubMed
Close
,
Gérard Maruani Assistance Publique-Hôpitaux de Paris, Hôpital Européen Georges Pompidou, Service de Physiologie, Paris, France
Centre de Référence des Maladies Rares du Calcium et du Phosphore Filière de Santé Maladies Rares OSCAR, Paris, France
Assistance Publique-Hôpitaux de Paris, Institut Necker-Enfants Malades, INSERM U1151 – CNRS UMR 8253, Paris, France

Search for other papers by Gérard Maruani in
Google Scholar
PubMed
Close
,
Eric Mirallie Chirurgie Cancérologique, Digestive et Endocrine, Institut des Maladies de l’Appareil Digestif, Hôtel Dieu, CHU Nantes, France
Association Francophone de Chirurgie Endocrinienne (AFCE), France

Search for other papers by Eric Mirallie in
Google Scholar
PubMed
Close
,
François Pattou Université de Lille, CHU Lille, Institut Pasteur Lille, Inserm U1190, Lille, France

Search for other papers by François Pattou in
Google Scholar
PubMed
Close
,
Riyad N H Seervai Molecular & Cellular Biology Graduate Program, Medical Scientist Training Program, Baylor College of Medicine, Houston, Texas, USA

Search for other papers by Riyad N H Seervai in
Google Scholar
PubMed
Close
,
Coralie Sido Hypoparathyroïdisme France, Annecy, France

Search for other papers by Coralie Sido in
Google Scholar
PubMed
Close
,
Caroline Silve Centre de Référence des Maladies Rares du Calcium et du Phosphore Filière de Santé Maladies Rares OSCAR, Paris, France
Assistance Publique-Hôpitaux de Paris, Hôpital Cochin, Biochimie et Génétique Moléculaires, Paris, France
INSERM, U1169, Université Paris Sud, Hôpital Bicêtre, Le Kremlin Bicêtre, France

Search for other papers by Caroline Silve in
Google Scholar
PubMed
Close
,
Aurélie Vilfaillot Assistance Publique-Hôpitaux de Paris, Hôpital Européen Georges Pompidou, Unité de Recherche Clinique, Paris, France
INSERM, U1418, CIC-EC, Hôpital Européen Georges Pompidou, Paris, France

Search for other papers by Aurélie Vilfaillot in
Google Scholar
PubMed
Close
,
Antoine Tabarin Service Endocrinologie Diabète et Nutrition, CHU de Bordeaux, Université de Bordeaux, Pessac, France

Search for other papers by Antoine Tabarin in
Google Scholar
PubMed
Close
,
Marie-Christine Vantyghem CHU Lille, Department of Endocrinology, Diabetology and Metabolism, Inserm U1190, EGID, Lille, France

Search for other papers by Marie-Christine Vantyghem in
Google Scholar
PubMed
Close
,
Pascal Houillier Assistance Publique-Hôpitaux de Paris, Hôpital Européen Georges Pompidou, Service de Physiologie, Paris, France
Centre de Référence des Maladies Rares du Calcium et du Phosphore Filière de Santé Maladies Rares OSCAR, Paris, France
Centre de Recherche des Cordeliers, INSERM, Sorbonne Université, Université de Paris, INSERM, UMRS1138, Paris, France
CNRS, ERL8228, Paris, France

Search for other papers by Pascal Houillier in
Google Scholar
PubMed
Close
, and
the investigators of the Épi-Hypo study
Search for other papers by the investigators of the Épi-Hypo study in
Google Scholar
PubMed
Close

Context

Recent guidelines have provided recommendations for the care of patients with chronic hypoparathyroidism. Very little is known about actual physicians’ practices or their adherence to such guidelines.

Objective

To describe the physicians’ practice patterns and their compliance with international guidelines.

Design

The cohort studies included were Épi-Hypo (118 physicians and 107 patients, from September 2016 to December 2019) and ePatients (110 patients, November 2019).

Methods

Internet-based cohorts involving all settings at a nationwide level (France). Participants were (i) physicians treating patients with chronic hypoparathyroidism and patients with chronic hypoparathyroidism either participating in the (ii) Épi-Hypo study (Épi-Hypo 2019 patients), or (iii) Hypoparathyroidism France, the national representative association (ePatients).

Results

The physicians’ specialties were mainly endocrinology (61%), nephrology (28%), family medicine (2.5%), pediatrics (2.5%), rheumatology (2%), or miscellaneous (4%) and 45% were practicing in public universities. The median number of pharmaceutical drug classes prescribed was three per patient. The combination of active vitamin D and calcium salt was given to 59 and 58% of ePatients and Épi-Hypo 2019 patients, respectively. Eighty-five percent of ePatients and 87% of physicians reported monitoring plasma calcium concentrations at a steady state at least twice a year. In 32 and 26% of cases, respectively, ePatients and physicians reported being fully in accordance with international guidelines that recommend targeting symptoms, plasma calcium and phosphate values, and urine calcium excretion.

Conclusions

The care of patients with chronic hypoparathyroidism involves physicians with very different practices, so guidelines should include and target other specialists as well as endocrinologists. Full adherence to the guidelines is low in France.

Open access
Marcela Moraes Mendes Department of Nutrition, Faculty of Health Sciences, University of Brasília, Distrito Federal, Brazil
Department of Nutrition, Institute of Life Sciences, Federal University of Juiz de Fora, Governador Valadares, Minas Gerais, Brazil
Department of Nutrition, Faculty of Health and Medical Sciences, University of Surrey, University of Surrey, Guildford, UK

Search for other papers by Marcela Moraes Mendes in
Google Scholar
PubMed
Close
,
Patricia Borges Botelho Department of Nutrition, Faculty of Health Sciences, University of Brasília, Distrito Federal, Brazil

Search for other papers by Patricia Borges Botelho in
Google Scholar
PubMed
Close
, and
Helena Ribeiro Department of Environmental Health, Faculty of Public Health, University of São Paulo, São Paulo, Brazil

Search for other papers by Helena Ribeiro in
Google Scholar
PubMed
Close

Vitamin D enhances calcium absorption and bone mineralisation, promotes maintenance of muscle function, and is crucial for musculoskeletal health. Low vitamin D status triggers secondary hyperparathyroidism, increases bone loss, and leads to muscle weakness. The primary physiologic function of vitamin D and its metabolites is maintaining calcium homeostasis for metabolic functioning, signal transduction, and neuromuscular activity. A considerable amount of human evidence supports the well-recognised contribution of adequate serum 25-hydroxyvitamin D concentrations for bone homeostasis maintenance and prevention and treatment strategies for osteoporosis when combined with adequate calcium intake. This paper aimed to review the literature published, mainly in the last 20 years, on the effect of vitamin D and its supplementation for musculoskeletal health in order to identify the aspects that remain unclear or controversial and therefore require further investigation and debate. There is a clear need for consistent data to establish realistic and meaningful recommendations of vitamin D status that consider different population groups and locations. Moreover, there is still a lack of consensus on thresholds for vitamin D deficiency and optimal status as well as toxicity, optimal intake of vitamin D, vitamin D supplement alone as a strategy to prevent fractures and falls, recommended sun exposure at different latitudes and for different skin pigmentations, and the extra skeletal effects of vitamin D.

Open access
Julia Herteux Medical University of Graz, Auenbruggerplatz, Graz, Austria

Search for other papers by Julia Herteux in
Google Scholar
PubMed
Close
,
Simon Johannes Geiger Medical University of Graz, Auenbruggerplatz, Graz, Austria

Search for other papers by Simon Johannes Geiger in
Google Scholar
PubMed
Close
,
Christina Starchl Medical University of Graz, Auenbruggerplatz, Graz, Austria

Search for other papers by Christina Starchl in
Google Scholar
PubMed
Close
,
Johanna Windisch Medical University of Graz, Auenbruggerplatz, Graz, Austria

Search for other papers by Johanna Windisch in
Google Scholar
PubMed
Close
,
Theresa Lerchl Medical University of Graz, Auenbruggerplatz, Graz, Austria

Search for other papers by Theresa Lerchl in
Google Scholar
PubMed
Close
,
Adelina Tmava-Berisha Medical University of Graz, Auenbruggerplatz, Graz, Austria

Search for other papers by Adelina Tmava-Berisha in
Google Scholar
PubMed
Close
,
Gerit Wünsch Medical University of Graz, Auenbruggerplatz, Graz, Austria

Search for other papers by Gerit Wünsch in
Google Scholar
PubMed
Close
,
Kathrin Eller Medical University of Graz, Auenbruggerplatz, Graz, Austria

Search for other papers by Kathrin Eller in
Google Scholar
PubMed
Close
,
Astrid Fahrleitner-Pammer Medical University of Graz, Auenbruggerplatz, Graz, Austria

Search for other papers by Astrid Fahrleitner-Pammer in
Google Scholar
PubMed
Close
, and
Karin Amrein Medical University of Graz, Auenbruggerplatz, Graz, Austria

Search for other papers by Karin Amrein in
Google Scholar
PubMed
Close

Objective

Chronic hypoparathyroidism (HP) is associated with acute and chronic complications, especially those related to hypocalcemia. We aimed to analyze details on hospital admissions and the reported deaths in affected patients.

Design and methods

In a retrospective analysis, we reviewed the medical history of 198 patients diagnosed with chronic HP over a continuous period of up to 17 years at the Medical University Graz.

Results

The mean age in our mostly female cohort (70.2%) was 62.6 ± 18.7 years. The etiology was predominantly postsurgical (84.8%). About 87.4% of patients received standard medication (oral calcium/vitamin D), 15 patients (7.6%) used rhPTH1–84/Natpar® and 10 patients (4.5%) had no/unknown medication. Two hundred and nineteen emergency room (ER) visits and 627 hospitalizations were documented among 149 patients, and 49 patients (24.7%) did not record any hospital admissions. According to symptoms and decreased serum calcium levels, 12% of ER (n = 26) visits and 7% of hospitalizations (n = 44) were likely attributable to HP. A subgroup of 13 patients (6.5%) received kidney transplants prior to the HP diagnosis. In eight of these patients, parathyroidectomy for tertiary renal hyperparathyroidism was the cause of permanent HP. The mortality was 7.8% (n = 12), and the causes of death appeared to be unrelated to HP. Although the awareness for HP was low, calcium levels were documented in 71% (n = 447) of hospitalizations.

Conclusions

Acute symptoms directly related to HP did not represent the primary cause of ER visits. However, comorbidities (e.g. renal/cardiovascular diseases) associated with HP played a key role in hospitalizations and deaths.

Significance statement

Hypoparathyroidism (HP) is the most common complication after anterior neck surgery. Yet, it remains underdiagnosed as well as undertreated, and the burden of disease and long-term complications are usually underestimated. There are few detailed data on emergency room (ER) visits hospitalizations and death in patients with chronic HP, although acute symptoms due to hypo-/hypercalcemia are easily detectable. We show that HP is not the primary cause for presentation but that hypocalcemia is a typical laboratory finding (when ordered) and thus may contribute to subjective symptoms. Patients often present with renal/cardiovascular/oncologic illness for which HP is known to be a contributing factor. A small but very special group (n = 13, 6.5%) are patients after kidney transplantations who showed a high ER hospitalization rate. Surprisingly, HP was never the cause for their frequent hospitalizations but rather the result of chronic kidney disease. The most frequent cause for HP in these patients was parathyroidectomy due to tertiary hyperparathyroidism. The causes of death in 12 patients appeared to be unrelated to HP, but we found a high prevalence of chronic organ damages/comorbidities related to it in this group. Less than 25% documented HP correctly in the discharge letters, which indicates a high potential for improvement.

Open access
Cristina Lamas Department of Endocrinology and Nutrition, Complejo Hospitalario Universitario de Albacete, Albacete, Spain

Search for other papers by Cristina Lamas in
Google Scholar
PubMed
Close
,
Elena Navarro Department of Endocrinology and Nutrition, Hospital Universitario Virgen del Rocío, Sevilla, Spain

Search for other papers by Elena Navarro in
Google Scholar
PubMed
Close
,
Anna Casterás Department of Endocrinology and Nutrition, Hospital Vall d’Hebron, Barcelona, Spain

Search for other papers by Anna Casterás in
Google Scholar
PubMed
Close
,
Paloma Portillo Department of Endocrinology and Nutrition, Hospital Clínico Universitario Virgen de la Arrixaca, Murcia, Spain

Search for other papers by Paloma Portillo in
Google Scholar
PubMed
Close
,
Victoria Alcázar Department of Endocrinology and Nutrition, Hospital Universitario Severo Ochoa, Leganés, Spain

Search for other papers by Victoria Alcázar in
Google Scholar
PubMed
Close
,
María Calatayud Department of Endocrinology and Nutrition, Hospital Univeristario Doce de Octubre, Madrid, Spain

Search for other papers by María Calatayud in
Google Scholar
PubMed
Close
,
Cristina Álvarez-Escolá Department of Endocrinology and Nutrition, Hospital Universitario La Paz, Madrid, Spain

Search for other papers by Cristina Álvarez-Escolá in
Google Scholar
PubMed
Close
,
Julia Sastre Department of Endocrinology and Nutrition, Complejo Hospitalario de Toledo, Hospital Virgen de la Salud, Toledo, Spain

Search for other papers by Julia Sastre in
Google Scholar
PubMed
Close
,
Evangelina Boix Department of Endocrinology and Nutrition, Hospital General Universitario de Elche, Elche, Spain

Search for other papers by Evangelina Boix in
Google Scholar
PubMed
Close
,
Lluis Forga Department of Endocrinology and Nutrition, Complejo Hospitalario de Navarra, Hospital de Navarra, Pamplona, Spain

Search for other papers by Lluis Forga in
Google Scholar
PubMed
Close
,
Almudena Vicente Department of Endocrinology and Nutrition, Complejo Hospitalario de Toledo, Hospital Virgen de la Salud, Toledo, Spain

Search for other papers by Almudena Vicente in
Google Scholar
PubMed
Close
,
Josep Oriola Biochemistry and Molecular Genetics Department, Hospital Clínic i Universitari de Barcelona, Barcelona, Spain

Search for other papers by Josep Oriola in
Google Scholar
PubMed
Close
,
Jordi Mesa Department of Endocrinology and Nutrition, Hospital Vall d’Hebron, Barcelona, Spain

Search for other papers by Jordi Mesa in
Google Scholar
PubMed
Close
, and
Nuria Valdés Department of Endocrinology and Nutrition, Hospital Universitario Central de Asturias, Oviedo, Spain

Search for other papers by Nuria Valdés in
Google Scholar
PubMed
Close

Primary hyperparathyroidism is the most frequent manifestation of multiple endocrine neoplasia type 1 (MEN1) syndrome. Bone and renal complications are common. Surgery is the treatment of choice, but the best timing for surgery is controversial and predictors of persistence and recurrence are not well known. Our study describes the clinical characteristics and the surgical outcomes, after surgery and in the long term, of the patients with MEN1 and primary hyperparathyroidism included in the Spanish Registry of Multiple Endocrine Neoplasia, Pheochromocytomas and Paragangliomas (REGMEN). Eighty-nine patients (49 men and 40 women, 34.2 ± 13 years old) were included. Sixty-four out of the 89 underwent surgery: a total parathyroidectomy was done in 13 patients, a subtotal parathyroidectomy in 34 and a less than subtotal parathyroidectomy in 15. Remission rates were higher after a total or a subtotal parathyroidectomy than after a less than subtotal (3/4 and 20/22 vs 7/12, P < 0.05), without significant differences in permanent hypoparathyroidism (1/5, 9/23 and 0/11, N.S.). After a median follow-up of 111 months, 20 of the 41 operated patients with long-term follow-up had persistent or recurrent hyperparathyroidism. We did not find differences in disease-free survival rates between different techniques, patients with or without permanent hypoparathyroidism and patients with different mutated exons, but a second surgery was more frequent after a less than subtotal parathyroidectomy.

Open access